Safety and Clinical Outcome of Bleomycin-Electrosclerotherapy (BEST) Treating Lymphatic Malformations (LMs)
Florian Obereisenbuchner, Elena Borisch, Daniel Puhr‐Westerheide, Sinan Deniz, Ferdi Çay, Mirjam Schirren, Gloria Biechele, Robin Schregle, Beate Häberle, Julia Haehl, A. Hartl, Alexandra Fröba-Pohl, Fatemeh Kashani, Jens Ricke, Max Seidensticker, Moritz Wildgruber, Vanessa F. Schmidt
Abstract
Abstract Introduction Bleomycin-electrosclerotherapy (BEST) is a novel treatment for slow-flow vascular malformations (SFVMs), most studied in venous malformations. This study specifically evaluated its safety and clinical outcome in lymphatic/lymphatic-dominant lympho-venous malformations (LMs/ld-LVMs). Materials and Methods A monocentric cohort with symptomatic LMs or ld-LVMs treated by BEST was retrospectively assessed. A treatment-specific, patient-reported questionnaire assessed overall clinical response (complete response: symptom-free, partial response: improved symptoms, no response: unchanged symptoms, progression of symptoms), subjective health-related quality of life (QoL; optimal, improved, unchanged, worsening), pain (numerical rating scale, NRS), and postprocedural skin discolouration (yes/no). Pre and postprocedural lesion size was measured in three planes on MRI. Results Twenty-seven treatments were performed in 20 patients with 14 LMs and six ld-LVMs (11 microcystic, five mixed, and four macrocystic subtypes). Patients received 1.4 ± 0.6 treatments with a median bleomycin dose of 7 mg (range 2–15 mg). After BEST, 7/20 (35%) patients reported complete response, 10/20 (50%) partial response, and 3/20 (15%) no response. Health-related QoL was stated as optimal in 10/20 (50%) and as improved in 4/20 (20%) patients. Median pain NRS was reduced from 6 (3–10) to 2 (0–6). Postprocedural skin discolouration occurred in 11/20 (55%) patients. Follow-up MR imaging revealed lesion size reduction from mean maximum volume of 793 cm 3 (IQR 155–2199 cm 3 ) to 548 cm 3 (IQR 71–1059 cm 3 ). Total complication rate (CIRSE grade 3–4) was 11.1%. No differences in all outcome parameters regarding LMs subtypes were assessed. Conclusion BEST demonstrates efficacy and acceptable safety treating LMs and ld-LVMs, including challenging microcystic lesions previously considered difficult to treat. Level of Evidence 3b, Retrospective Cohort Study. Graphical Abstract